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Eur J Cardiothorac Surg 2002;21:489-496
© 2002 Elsevier Science NL
a Department of Thoracic Surgery, School of Medicine, University of Selcuk, 42080 Meram-Konya, Turkey
b Department of Chest Diseases, School of Medicine, University of Selcuk, 42080 Meram-Konya, Turkey
c Department of Infectious Diseases, School of Medicine, University of Selcuk, 42080 Meram-Konya, Turkey
d Department of Emergency Medicine, School of Medicine, University of Selcuk, 42080 Meram-Konya, Turkey
Received 3 October 2001; received in revised form 3 December 2001; accepted 3 December 2001.
* Corresponding author. Tel.: +90-332-3232600, ext. 1844; fax: +90-332-3232643
e-mail: olgun{at}selcuk.edu.tr
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Abstract |
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 Top Abstract 1. Introduction2. Materials and methods3. Results4. DiscussionReferences |
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Key Words: Hydatid cyst • Liver • Lung • Hepatopulmonary • Surgical approaches
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1. Introduction |
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TopAbstract1. Introduction2. Materials and methods3. Results4. DiscussionReferences |
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Recently, it has been noticed that the number of cases with multi-organ localizations and multiple cysts has increased [5]. Abdominal echinococcosis — mostly hepatic cysts — accompanying pulmonary cysts constitutes most multi-organ localizations. There are only a few articles in literature dealing separately with such cases [5–8].
In this article, we aimed to evaluate the differences of clinical, radiological and surgical features, and to investigate the therapeutic strategies of the patients affected by single pulmonary (SPH) and hepatopulmonary hydatidosis (HPH).
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2. Materials and methods |
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TopAbstract1. Introduction2. Materials and methods3. Results4. DiscussionReferences |
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We evaluated and compared the groups with respect to age, sex, symptoms, interval between the onset of symptoms and hospitalization, laboratory findings, hemithorax and lobar localizations, and state, number and size of cysts, preoperative complications, surgical approaches and techniques, postoperative hospitalization and follow-up periods and postoperative morbidity and mortality.
All the patients underwent surgical interventions. The methods of surgical management have been outlined in the following section.
2.1. Thoracic approaches
All procedures were carried out under general anaesthesia with double-lumen endotracheal intubations. Although classical posterolateral thoracotomy was used in most of the patients, recently muscle-preserving thoracotomy has been preferred.
In Group I, thoracotomy in 91 patients and median sternotomy in one patient were performed. In Group II, thoracotomy was carried out in 34 patients (69.4%) and median sternotomy in one patient (2%). Hepatic cysts of these patients were treated via percutaneous drainage or laparotomy by associated clinics. Fourteen patients (28.6%) having concomitant cysts in the liver dome were managed via thoracophrenotomy.
The pleural space was routinely lavaged with hypertonic saline solution in all patients of both groups and intact cysts were isolated by hypertonic saline compresses to preserve the surrounding tissues.
2.2. Operative techniques
2.2.1. Pulmonary cysts
In both groups, hydatid fluid of the intact cysts was first aspirated from the top of the cyst with 20-gauge needle for the purpose of lowering intracystic pressure. Later, placing suction apparatus into the cyst from the same site ensured complete aspiration of the fluid. Germinative membrane was easily removed from the cyst by widening needle insertion site (cystotomy). Pericystectomy was preferred in cysts with superficial cavity in both groups. Enucleation due to the risk of rupture was not applied except in two patients in Group II.
Decortication was performed particularly in complicated cysts, which had opened into the pleura and so led to a severe pleural thickening in both groups. Later, residual cavity was carefully cleaned and controlled for the presence of additional daughter vesicles. Cystic space was irrigated with 1% povidine-iodine in complicated cysts. All bronchial openings were closed individually with absorbable sutures. The cavity was obliterated with purse-string absorbable sutures, starting from the bottom (capitonnage). Radical resections such as wedge resection, segmentectomy and lobectomy were carried out in cysts, which had caused irreversible changes in the adjacent lung parenchyma. The surgical intervention was ended following placement of chest tubes into the apical and the costophrenic localizations of the pleural space.
2.2.2. Hepatic cysts
The techniques, described above for pulmonary cysts, were applied in the similar way, following phrenotomy in Group II. Evident biliary openings were closed. The free parts of fibrous capsule were inverted or an omental flap was placed into the residual cavity. Pursuing that, subdiaphragmatic drainage was carried out and the diaphragm was closed.
2.3. Medical treatment
Mebendazole (50 mg/kg/day) or albendazole (10 mg/kg/day) for a probable recurrence was given to all patients in the postoperative period for at least 2 months.
2.4. Data analysis
Student's-t test in numerical data showing normal distribution and Mann–Whitney U test in conditions of abnormal distribution were used in the comparison between groups. Pearson Chi-square, Chi-square with Yates correction and Kolmogarov–Smirnov test were applied in the comparison of non-parametric data. P<0.05 was accepted to indicate significance.
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3. Results |
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TopAbstract1. Introduction2. Materials and methods3. Results4. DiscussionReferences |
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Sixty-three (68.5%) of 92 patients were male and 29 patients (31.5%) were female in Group I. There were 17 male (34.7%) and 32 female patients (65.3%) in Group II. Male patient ratio was higher in Group I whereas female patient ratio was higher in Group II and the difference between groups was significant (P<0.05). The age and sex distribution of the patients are shown in Table 1.
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3.4. Radiological examination
Frequently seen pulmonary radiological pathology was the homogenous density in both groups (Group I, 46.8% vs. Group II, 59.6%). These results are listed in Table 2. The water lily sign, cavitary image and pulmonary collapse were more common in the chest X-ray of the patients of Group I whereas the elevation of the diaphragm, pneumonic infiltration, the closure of the costodiaphragmatic sinus and the pleural thickening were more frequent in Group II. However, the frequency of air–fluid level and solitary pulmonary nodule was the same in both groups. The abdominal ultrasonographic examination of the patients in Group II revealed a total of 72 cysts, of which 75% was located in the right and 25% in the left liver lobe. Additional cysts were also detected in the spleen of four patients.
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3.6. Number of cysts
Although the majority (67.3%) of the cysts located in the liver was solitary, the frequency of solitary pulmonary cyst in Group I (77.2%) was higher than that in Group II (54.3%). However, the multiple cyst frequency in Group II (45.7%) was higher than the frequency in Group I (22.8%). The differences between groups were significant (P<0.05).
3.7. Localization
Of 92 patients in Group I, 48 patients (52.2%) had pulmonary cysts in the right, 32 (34.8%) in the left and 12 (13%) in bilateral hemithorax, whereas, of 49 patients in Group II, 32 patients (65.3%) had pulmonary cysts in the right, four (8.2%) in the left and 13 patients (26.5%) had cysts in the bilateral hemithorax (Fig. 2
). When the hemithorax distribution in both groups were compared, left hemithorax localization was significantly higher in Group I and bilateral hemithorax localization was higher in Group II (P<0.05).
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When the distribution of pulmonary cysts according to size was concerned, the frequency of the appearance of cysts with 2–5 cm diameter was 46.7% in Group I and 65.2% in Group II. Cysts with 6–9 cm diameter were equally distributed in both groups (21.7% in each group) and cysts larger than 10 cm were seen in 31.5 and 13% in Groups I and II, respectively. Pulmonary hydatid cysts smaller than 10 cm, particularly 2–5 cm in diameter, were significantly more predominant in Group II and cysts larger than 10 cm were significantly more predominant in Group I (P<0.05).
3.9. Preoperative complications
Complications were present at the time of presentation in 27 (29.3%) of 92 patients in Group I and 14 (28.6%) of 49 patients in Group II. These complications and percentages in groups are shown below: pneumothorax (Group I, 6.5% vs. Group II, 6.1%), empyema (Group I, 9.8% vs. Group II, 4.1%), pleural thickening (Group I, 12% vs. Group II, 8.2%), pericarditis (only in Group I, 1.1%), hepatopleural fistula (only in Group II, 8.2%) and hepatobronchial fistula (only in Group II, 2%).
3.10. Operation types
Lung saving surgical procedures (Group I, 87% vs. Group II, 94%) were used more than radical surgery (Group I, 13% vs. Group II, 6%). The most preferred intervention was cystotomy with capitonnage (Group I, 78% vs. Group II, 82%). Decortication was applied to patients who had severe pleural thickening (Group I, 25% vs. Group II, 20%). Operation types, employed in both groups, are shown in Table 3.
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The mean hospital stay of patients in Groups I and II were 9±4 and 10±6 days, respectively. The postoperative complication ratio and the hospital stay in Group II did not significantly differ from those in Group I.
3.12. Follow-up and recurrence
The mean follow-up periods were 27±14 months (range 4–58 months) in the patients of Group I, and 26±11 months in those of Group II (range 5–51 months). No recurrence was noted in the patients of both groups during this period.
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4. Discussion |
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TopAbstract1. Introduction2. Materials and methods3. Results4. DiscussionReferences |
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Lungs and the liver are the organs involved frequently in hydatid disease. The liver in adults and the lungs in children are the predominant sites [10]. It develops outside these organs in 2.1% of the patients [4]. Extrapulmonary intrathoracic localization is seen in 8% [11]. As reported in the literature, cysts in two-thirds of patients are unilateral and/or solitary [2,3,5,12,13]. However, recently it has been noted that there is an increase in the patients with multi-organ localization and multiple cysts [5]. Hydatidosis with multi-organ localization, though it is seen in variable organ combinations, expresses predominantly the presence of cysts both in lungs and liver. Such cysts, named hepatopulmonary hydatid cysts, were reported in 8.8–36.5% in different series [1,2,4,5,14]. This ratio was 34.8% in our series.
Single pulmonary localization of the hydatid disease is more common in children and youth than adults [3]. Although single pulmonary cyst in adults is seen in both sexes, it is more frequent in males (53–70%) in the second and third decades [2,10,15]. We found similar results in our patients with SPH (Group I) as well. Nevertheless, it was worth noting the higher presence of hepatopulmonary cysts especially in female adults more than 40 years of age.
Although pulmonary cysts are asymptomatic in 8–32% of the patients with hydatid disease, the symptoms are generally related to the cyst size or are complicated [1–3]. The pulmonary cysts may grow fast due to the elastic structure of the lungs and they may reach huge sizes especially in children. In that occasion, they may cause the symptoms such as cough, chest pain, dyspnea, expectoration and hemoptysis [2,6,12,16]. However, the cysts may grow slowly in the liver because of the organ solidity [1,13,16]. In our series we found no difference between the mean diameters of hepatic and pulmonary hydatid cysts. This might be due to the huge cysts located at the dome of liver since these cysts may reach huge sizes similar to the cysts in pulmonary localization. When the pulmonary cysts were concerned, the cysts less than 10 cm, mainly 2–5 cm in size were significantly higher in the patients with HPH (Group II) whereas those more than 10 cm were higher in the patients of Group I. Parallel to that, the symptoms even in symptomatic patients of HPH were pulmonary in origin and they included cough, chest pain, dyspnea, expectoration and hemoptysis. The gastrointestinal symptoms such as abdominal pain, biloptysis, nausea, vomiting, loss of appetite and fatigue were very rare. This finding does warrant the examination of liver involvement in all patients with pulmonary cysts.
Although pulmonary cysts may establish in every lobe of lungs, they are more frequent in lower lobes and mainly in the right hemithorax [1–3,12,15,16]. The hepatic cysts also prefer the right lobe (73%) of liver [1,7]. Our findings were consistent with the literature in such a manner that the pulmonary cysts in Group II were abundant in the right lung (62.5%) and the lower lobes (41% in right and 16% in left) and the hepatic cysts in the right lobe in 75% of the patients. Although the majority of the pulmonary cysts are unilateral and solitary [2,5,13,17], bilateral localization is reported in 2–30% [1,3,12,13]. In our series bilateral pulmonary involvement was significantly higher in Group II (26.5%), compared to Group I (13%). In addition, multiplicity was very high and demonstrated almost in half of patients (45.7%) of Group II. The ratio of the multiple cysts was 22.8% in Group I. This might be the result of a spreading due to a rupture of the hepatic cysts, known to have more daughter vesicles than pulmonary cysts [1]. Petrov et al. [5] noticed an increase in cases with multiple cysts and multi-organ localizations since 1988. So, staged operations and thus prolonged care undertaken in the patients with three localizations of the hydatid disease (bilateral lungs+liver) may cause an additional economic and labour loss. Recently, a variety of operative strategies and approaches have become very popular to decrease them to a minimum. Particularly bilateral thoracophrenotomy or sternophrenotomy, and sternolaparotomy procedures have been increased gradually, making it possible to operate in one session [5,6,8,18].
Although tests such as the Casoni's skin test, the Weinberg haemagglutination test, the eosinophils count and the sedimentation rate were reported to be more useful in ruptured cysts [2], nowadays they have not been routinely used due to their low diagnostic efficiencies [1,17]. The most important diagnostic tool in hydatid cysts is chest X-ray [1,11,15,17]. It is typical for an intact cyst to present as round or oval homogenous densities with sharp contours. Computed tomography of the chest clearly reveals especially cysts, which are complicated, behind or in the heart or mediastinum [11,15,19]. Ultrasonography and echocardiography are two indisputable methods to evaluate the hepatic and/or pericardiac–cardiac cysts [11]. Nearly 1/2 or 1/3 of cases with hydatid cysts are complicated [1]. The majority of the pulmonary cysts in our series was complicated in both groups (Group I, 59.8% vs. Group II, 56.5%). Hydatid cysts, when they rupture, usually open into a bronchus and the patients may describe this phenomenon as salty or peppery water expectoration which means spring water expectoration and sometimes patients may expectorate membranous particles like egg white. In our series, salty water and membranous expectoration were seen in 13.8% of the patients with SPH and in 12.8% of the patients with HPH.
A rupture of the cyst into the pleural cavity is a severe complication but rarely seen and is reported 0.5–18.2% in the literature [13,14,20,21]. This complication may be associated with both pulmonary and hepatic cysts [1,2,4,13,14,20,21]. This rarity is due to the dense adhesions between the cyst and the parietal pleura, not letting the cysts to open into the pleural cavity [20]. Such dense severe pleural adhesion and thickening were evident in 12 and 8.2% of the patients of Groups I and II, respectively.
In our series, the pleural complications were present in 29.3% of the patients in Group I and 28.6% of the patients in Group II. It is deemed that the pleural necrosis development due to the pressure of pulmonary cysts, especially in peripheral and subpleural localizations have important role in the opening of cysts into the pleural cavity. The most common pleural complications noted in our series were empyema (Group I, 9.8% vs. Group II, 4.1%) and pneumothorax (Group I, 6.5% vs. Group II, 6.1%). It is widely accepted that the liver dome cysts may rupture into the pleural cavity because of the irritative effect of bile and the pressure upon the diaphragm. We demonstrated hepatothoracic complications in 10.2% of the patients with HPH.
The treatment of the pulmonary hydatid cysts is primarily surgical intervention [1–21]. The aim of surgery is mainly the removal of the germinative membrane without allowing intraoperative seeding and the prevention of the intrapulmonary residual cystic space. A number of factors and variables should be taken into consideration in the management of hydatid cysts. Thus, the flexibility is important in decision making about the type of operation [22]. Lung saving surgeries such as enucleation, pericystectomy, simple cystotomy and with or without the capitonnage of the pericystic space are usually preferred as a first choice [1,2,8,12,15,16]. Radical resection surgery should be limited only to the infected cysts, which had caused the irreversible changes in the surrounding pulmonary parenchyma. The priority should be given to lung saving surgeries even in the patients with giant hydatid cysts although the difficulties in the re-expansion of the lung are due to the prolonged pressure of the cyst upon the lung parenchyma [12,15,16]. We performed lung saving surgeries in most of our patients. Cystotomy with capitonnage was the procedure frequently chosen in both groups (Group I, 78% vs. Group II, 82%). Radical resections were undertaken in 13 and 6% of the patients of Groups I and II, respectively.
Postoperative complications such as prolonged air leak, empyema, pneumonia and atelectasis have been reported in 1.4–19.1% and mortality in 0–2% in different series [1,2,4,15]. No significant differences have been noted between radical and lung saving surgeries about the postoperative complications in the literature [2,15,16]. In parallel to that, we met postoperative complications in 17% of the patients with SPH and in 16% of the patients with HPH. Only one patient (0.7%) was lost due to the cardiac problems in Group I, however, no exitus was seen in Group II. Although the local recurrence rate is somewhat higher after lung saving surgeries, it has been reported in 0–11% in different series. Most of the local recurrences have been observed within the first year of the operation [1,4,5,12,15,16]. We did not witness any recurrences in our patients, who had been followed for about a mean of 2.5 years.
As it is well known, the daughter vesicles in the cyst fluid are found in 40% of the hepatic cysts. This may lead to a preoperative secondary pulmonary cyst development or a postoperative local recurrence due to the spillage of daughter vesicles into the operation field [23]. Thus, radical resection has been recommended in the hepatic cysts [24]. Although we did not carry out radical resection for the hepatic cysts, there was no recurrence in our patients. This might be due to the postoperative chemotherapy. Successful results with chemotherapy have also been noted in the prevention of postoperative local recurrences in the literature [1,15].
It has been reported that the postoperative complications are more frequent and severe in hepatic rather than in pulmonary cysts [1,8,22], however, we did not determine any significant difference between groups for both postoperative complications and hospital stay. In addition to that, the morbidity and the mortality ratios did not differ significantly either. These findings emphasize the importance of the operation approaches, rather than the types. Therefore, it is obvious that due to the staged operations, the economic and labour loss will be much more in multi-organ, compared to single organ involvement. For this purpose, surgical approaches such as bilateral successful thoracotomy, Clamshell incision bilateral thoracotomy or sternotomy has been carried out in the patients with bilateral pulmonary hydatid cysts. Recently, one-session operation has been recommended in the patients having hydatid cysts with multi-organ localizations, as well [5,6]. The strategy in one-session operation is to use the way of right thoracophrenotomy in the patients having concomitant right pulmonary cysts, and to use sternophrenotomy or sternolaparotomy in the patients with concomitant bilateral pulmonary cysts. In our series, among 49 patients with HPH, 32 (65.3%) were suitable for right thoracophrenotomy and 13 (26.5%) patients for sternophrenotomy. However, right thoracophrenotomy could be performed only in 14 of these 32 patients who had cysts in the hepatic dome. In the remaining 18 patients this approach could not be carried out since the hepatic cysts were not reachable. It has been reported that sternotomy is contraindicated if hydatidosis is associated with pleural complications and/or if the cysts are massive or infected and/or if the radical resection (especially left lower lobectomy) is mandatory [5,6]. These conditions further limit the usability of this approach. In our series, pleural complications were present in 28.6% of the patients with HPH and 56.5% of the pulmonary cysts were suppurated at the time of admission. Additionally, 6% of the cysts could only be managed via radical pulmonary resection. In the light of these findings we believe that the sternotomy could only be appropriate in selected cases although Cetin et al. [8] have suggested the management of the hepatic cysts in another session.
In conclusion, HPH is more frequent in female patients and particularly in those over 40 years of age. The pulmonary cysts in such patients have a tendency to be bilateral and multiple. Thus, they differ from single pulmonary cyst in such clinical features. Since the management in these patients may lead to substantial economic and labour loss because of the multi-operations and prolonged postoperative care, the therapeutic approach and the policy should be different in these patients to minimize the loss. For this purpose, the right way to manage such patients is to use thoracophrenotomy in the patients having right pulmonary cyst concomitant with especially hepatic dome cysts, or to use sternal approach in the patients with bilateral pulmonary cysts associated with the hepatic cysts, after elimination of the contraindications.
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References |
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TopAbstract1. Introduction2. Materials and methods3. Results4. DiscussionReferences |
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ahin A. Usually located hydatid cysts: intrathoracic but extrapulmonary. Ann Thorac Surg 1997;64:334-337.
tepe I., Bozkurt D., Gülhan E., Çetin G. Surgical treatment of pulmonary hydatid cysts in children. J Thorac Cardiovasc Surg 2000;120:1097-1101.This article has been cited by other articles:
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  M. Aghajanzadeh, F. Safarpoor, H. Amani, and A. Alavi One-Stage Procedure for Lung and Liver Hydatid Cysts Asian Cardiovasc Thorac Ann, October 1, 2008; 16(5): 392 - 395. [Abstract] [Full Text] [PDF]
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 S. J. Santivanez, A. E. Sotomayor, J. C. Vasquez, J. G. Somocurcio, S. Rodriguez, A. E. Gonzalez, R. H. Gilman, H. H. Garcia, and the Cysticercosis Working Group in Peru Absence of Brain Involvement and Factors Related to Positive Serology in a Prospective Series of 61 Cases with Pulmonary Hydatid Disease Am J Trop Med Hyg, July 1, 2008; 79(1): 84 - 88. [Abstract] [Full Text] [PDF]
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 S. Halezeroglu Resection of intrathoracic and subdiaphragmatic hydatid cysts MMCTS, April 25, 2005; 2005(0425): 307. [Abstract] [Full Text] [PDF]
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 E. Sahin, S. Enon, A. K. Cangir, H. Kutlay, S. Kavukcu, H. Akay, I. Okten, and S. Yavuzer Single-stage transthoracic approach for right lung and liver hydatid disease J. Thorac. Cardiovasc. Surg., September 1, 2003; 126(3): 769 - 773. [Abstract] [Full Text] [PDF]
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